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Clinically important age-related differences in sleep related disordered breathing in infants and children with

Michal Cohen¹, Jill Hamilton¹, Indra Narang²

¹Division of Endocrinology, The Hospital for Sick Children, Toronto, Ontario, Canada; The University of Toronto, Toronto, Ontario, Canada.

|July 1, 2014

Insights

Infants with Prader-Willi Syndrome (PWS) show higher rates of central sleep apnea (CSA) than older children. Supplemental oxygen effectively treats CSA in infants with PWS, improving oxygen saturation.

Area of Science:

Pediatric Pulmonology
Sleep Medicine
Genetics

Background:

Sleep-related disordered breathing (SDB) in Prader-Willi Syndrome (PWS) is increasingly recognized.
The link between growth hormone therapy, SDB, and sudden death in PWS warrants further investigation.
Limited data exists on SDB patterns and management in infants with PWS.

Purpose of the Study:

To describe SDB patterns in growth hormone-naive infants with PWS.
To outline the management of SDB in infants with PWS at the institution.
To compare sleep disorder patterns between infants and children with PWS.

Main Methods:

Retrospective review of polysomnograms from 44 patients (0-18 years) with PWS.
Analysis of age, sex, anthropometrics, sleep architecture, apnea indices, and oxygen saturation.
Evaluation of oxygen therapy efficacy in infants with central sleep apnea.

Main Results:

Infants (under 2 years) had significantly higher rates of central sleep apnea (43%) compared to older children (5%).
Obstructive sleep apnea was more prevalent in older children.
Supplemental oxygen significantly reduced central apnea index and improved oxygen saturation in treated infants.

Conclusions:

Central sleep apnea is more common in infants with PWS than in older children.
Supplemental oxygen is an effective treatment for central sleep apnea in infants with PWS.
Routine sleep surveillance and oxygen treatment for CSA in PWS patients are recommended.

Read full analysis

Abstract

Background:

Sleep related disordered breathing (SDB) in pediatric Prader-Willi Syndrome is gaining increased attention due to the possible association of growth hormone therapy, SDB and sudden death. However data on the patterns of SDB and their management, particularly in infants in this population, is lacking.

Objective:

The aim of this study was to 1) describe patterns of SDB in growth hormone naive infants with PWS and the management of these disorders in our institution 2) Compare the patterns of sleep disorders between infants and children with PWS.

Methods And Design:

Polysomnograms of infants and children (0-18 years of age) with Prader-Willi Syndrome were reviewed. Age, sex, anthropometrics, sleep architecture, obstructive and central apnea indices and oxygen saturations were recorded. Data of infants with central sleep apnea treated with oxygen were analyzed to evaluate the efficacy of this treatment. The main outcome measures were obstructive and central apnea indices on a polysomnogram.

Results:

Data of 44 patients, 23 under 2 years of age and 21 older children were included. Infants when compared with older children were more likely to experience central sleep apnea (43% vs. 5%; p = 0.003). In older children obstructive was significantly more prevalent than central sleep apnea. Supplemental oxygen was used to treat 9/23 infants with central sleep apnea. Oxygen therapy resulted in a significant decrease in the median central apnea index from 14 (5,68) to 1 (0,6; p = 0.008) events/hour and an improvement in the oxygen saturation nadir from 70% (52, 92) to 81% (64, 95; p = 0.080).

Conclusions:

Central sleep apnea with associated oxygen desaturations is more prevalent in infants compared with older children with Prader-Willi Syndrome. Supplemental oxygen was efficacious in treating central sleep apnea in infants. Routine sleep surveillance for all children with Prader-Willi Syndrome and treatment with oxygen for central sleep apnea should be considered.

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